Extreme subcutaneous insulin resistance is a rare syndrome characterized by severe resistance to subcutaneous insulin with normal intravenous insulin sensitivity. It is pathophysiology is unknown, though an increased insulin degrading activity has been suggested [1]. We report a case of extreme insulin resistance in a stable, diabetic patient without any acute complication related to diabetes mellitus. A 6 year old diabetic patient, from Ardabil, that presented with unusually uncontrolled diabetes mellitus due to extreme insulin resistance. That finally our patient was treated by Lantus, Apidra and Rituximab.